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Ameloblastic Fibro-Odontoma 1Valappil T Beena, 2Jasmin Jose, 3Sabu Paul, 4Manju S Mathunny, 5Sreela K Kottarathil, 6Shalu

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Ameloblastic Fibro-Odontoma 1Valappil T Beena, 2Jasmin Jose, 3Sabu Paul, 4Manju S Mathunny, 5Sreela K Kottarathil, 6Shalu OMPJ Valappil T Beena et al 10.5005/jp-journals-10037-1108 CASE REPORT Ameloblastic Fibro-odontoma 1Valappil T Beena, 2Jasmin Jose, 3Sabu Paul, 4Manju S Mathunny, 5Sreela K Kottarathil, 6Shalu ABSTRACT lesions with odontogenic epithelium with odontogenic 1 Introduction: Ameloblastic fibro-odontoma (AFO) is a rare ectomesenchyme, with or without hard tissue formation. benign, slow-growing, expansile epithelial odontogenic tumor AFO is a tumor with histological features similar to those with odontogenic ectomesenchyme. It constitutes about 1 to of ameloblastic fibroma (AF), but it shows inductive 3% of all odontogenic tumors. This lesion usually occurs in changes that lead to the formation of dentin or enamel. people less than 20 years old, and thus age is an important characteristic in the differential diagnosis. It may inhibit tooth eruption or displace involved teeth, although teeth in the affected area are vital.2-4 The lesions Case report: An 8-year-old boy reported with a swelling in the right mandibular region. Panoramic radiograph showed are usually diagnosed during the first and second decades a well-demarcated radiolucency containing radiopaque areas of life. Radiographically, the lesion presents with a well- of irregular shape extending from the right deciduous second circumscribed, unilocular or multilocular radiolucent molar to the permanent third molar region. A provisional clini- area containing various amounts of radiopaque material cal diagnosis of complex odontome was made. Ameloblastic fibro-odontoma, odontoameloblastoma, calcifying epithelial of irregular size and form. The large areas of calcification odontogenic tumor, and calcifying odontogenic cyst were also make it impossible to radiographically differentiate it included in the differential diagnoses. from complex odontoma.4,5,6 This article reports a case of Management and prognosis: Patient underwent tumor exci- AFO in an 8-year-old male with significant clinicoradio- sion, and histopathological examination confirmed as AFO. The graphic features, histopathology, differential diagnoses, patient was followed postoperatively for 12 months and no sign and treatment. of recurrence was noted. Conclusion: Long-term follow-up at regular short intervals CASE REPORT should be maintained in the management of AFO. Here we add one more case to the sparsely reported AFO with a brief An 8-year-old boy was referred to the outpatient depart- review of literature. ment at the Government Dental College, Kottayam, Keywords: Ameloblastic fibro-odontoma, Odontogenic tumors, Kerala, India, with a swelling in the right mandibular Odontoma. region. How to cite this article: Beena VT, Jose J, Paul S, Mathunny MS, There was no significant medical, social, or family Kottarathil SK, Shalu. Ameloblastic Fibro-odontoma. Oral history or history of local trauma or infection. Extraoral Maxillofac Pathol J 2017;8(2):94-97. examination showed mild facial asymmetry on the right Source of support: Nil side. On palpation, a hard painless swelling was observed in the right mandibular body. Intraoral examination Conflict of interest: None revealed a well-circumscribed swelling of the alveolar ridge, extending from the deciduous second molar to INTRODUCTION the anterior border of the ascending ramus, covered with Ameloblastic fibro-odontoma (AFO) is a rare benign, normal mucosa which was hard on palpation. The man- slow-growing, expansile epithelial odontogenic tumor dibular right permanent first molar was absent. with odontogenic ectomesenchyme. According to the Panoramic radiograph showed a large, well-demar- World Health Organization classification of odontogenic cated radiolucency containing tooth-like radiopaque tumors published in 2005, AFO belongs to the group of areas of irregular shape extending from the right decidu- ous second molar to the permanent third molar region. The tooth germ of the right lower permanent first molar was displaced to the base of the mandible by the lesion. 1Professor and Head, 2,5,6Postgraduate Student, 3Assistant Professor, 4Senior Lecturer The right lower permanent second and third molar tooth germs were involved by the mineralized mass (Fig. 1). 1-6Department of Oral Pathology and Microbiology, Government Dental College, Kottayam, Kerala, India Going by the clinical and radiographic findings, a provi- sional clinical diagnosis of complex odontome was made. Corresponding Author: Valappil T Beena, Professor and Head, Department of Oral Pathology and Microbiology Ameloblastic fibro-odontoma, odontoameloblastoma, Government Dental College, Kottayam, Kerala, India, Phone: calcifying epithelial odontogenic tumor, and calcifying +919847150407, e-mail: [email protected] odontogenic cyst were also included in the differential 94 OMPJ Ameloblastic Fibro-odontoma epithelium and loosely arranged spindle-shaped cells identical to stellate reticulum in the central area (Fig. 2). The ectomesenchymal component comprised loose connective tissue, with abundant extracellular matrix containing young, plump fibroblasts, sometimes with a stellate appearance, resembling the dental papilla. The decalcified sections showed enamel matrix and dentin-like substance (Fig. 3). In few areas, reduced enamel epithe- lium was observed around the matrix. The dental tissues were disorganized in some regions forming a large mass Fig. 1: Panoramic radiograph showing well-demarcated radiolucent of dentin around reduced enamel epithelium and enamel lesion affecting the right lower permanent molar region, containing matrix. No malignant changes were evident. Based on radiopaque areas of varying size, causing the displacement of the tooth germ of the lower first molar clinical and histopathological features, the tumor was diagnosed as AFO. No sign of recurrence was observed diagnoses. Under general anesthesia, by intraoral during a postoperative period of 12 months. approach, the tumor was excised. DISCUSSION Light microscopic examination of sections revealed strands and islands of odontogenic epithelium showing The present case meets the clinicopathological criteria for peripheral palisading columnar cells resembling enamel the diagnosis of AFO, which is a rare mixed odontogenic A B Figs 2A and B: Strands and islands of odontogenic epithelium showing peripheral palisading and loosely arranged central stellate reticulum-like cells embedded in myxoid cell-rich stroma resembling the dental papilla: (A) (hematoxylin and eosin [H&E] 100×); and (B) (H&E 400×) A B Figs 3A and B: Photomicrograph of the decalcified sections showing dentin-like material and enamel matrix around reduced enamel epithelium (A) (hematoxylin and eosin [H&E] 100×); (B) (H&E 400×) Oral and Maxillofacial Pathology Journal, July-December 2017;8(2):94-97 95 Valappil T Beena et al tumor. The AFO accounts for 1 to 3% of all odontogenic that different lesions are being described under the same tumors.5 The tumor has equal distribution in the maxilla term of AFO, where some are hamartomatous in nature and mandible and shows predilection to the molar region. and others a true de novo neoplasm. Currently, it is not There is no gender variance and mean age of subjects is possible to distinguish a hamartoma from a neoplasm 11.5 years. Slootweg7 reviewed 50 patients with this lesion, only based on histological features. A small lesion on for whom the mean age was 8.1 years. This lesion usually the occlusal surface of an unerupted molar may be the occurs in people less than 20 years old, and thus age is immature stage of an odontoma, whereas a lesion that an important characteristic in the differential diagnosis. causes swelling and cortical expansion is likely to be a The two frequent patient complaints are failure of true AFO.14 tooth eruption and swelling. Asymptomatic cases are inci- Immunohistochemistry reveals that all epithelial dentally detected on radiography. This lesion is generally components in the AF-like area showed expression of considered a slow-growing central jaw tumor; however, CK8, CK13, CK16, CK14, CK18, and CK19, and coex- several exceptions to this pattern have been reported.5 pression of these cytokeratins and vimentin. Miyauchi Radiographs reveal a well-defined radiolucent area et al14 suggest that the epithelial component showed cell enclosing varying amount of radiopaque material of differentiation of the enamel organ with the potential for irregular size and shape.8 The ratio of radiolucent to epithelial–mesenchymal induction. The cell kinetics in radiopaque areas ranges from one lesion to another. Some mandibular AFO was studied by bromo-deoxyuridine of the lesions are relatively small when first detected, and proliferating cell nuclear antigen. The result of measuring 1 to 2 cm in diameter, whereas others may be immunohistochemical investigations suggested that the exceedingly large, involving a considerable portion of the mesenchymal component was more proliferative than the body of the mandible or maxilla.7 epithelial component.15 Such an immunohistochemical Histologically, AFO is composed of cords, strands, investigation may be useful in evaluating the proliferative and islands of odontogenic epithelium within a cellular potential of odontogenic tumors. primitive ectomesenchyme that simulates dental papilla. Slootweg16 compared ultrastructure of the epithelial– Dentin and enamel matrix are also seen.9 Conversely, the mesenchymal interface of AFOs with that of the nor- elite presence of dentin depicts another entity, amelo- mally developing tooth germ and it was noted that blastic fibrodentinoma. The
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